Treatment with intravenous ganciclovir for six weeks prevents hearing deterioration in children with symptomatic congenital cytomegalovirus (CMV) infection. Prolonged treatment might be more beneficial, but is associated with the potential side-effects of long-term use of intravenous lines. Oral valganciclovir could be an alternative because of its excellent bio-availability, reaching plasma concentrations similar to those achieved with intravenous ganciclovir in neonates with symptomatic CMV infection. We present a two-month-old girl with congenital CMV infection with central nervous disease involvement, who was treated with intravenous ganciclovir for 15 days followed by oral valganciclovir (30 mg/kg/day in 2 doses) for 6 months. Treatment resulted in adequate ganciclovir plasma levels, suppressed plasma viral load, prevention of hearing deterioration and was well tolerated, with no apparent side-effects.